A 76-year-old woman with a 5-mo history of recurrent diarrhea and generalized edema was admitted to our hospital. Repeated colonoscopies and gastrointestinal scope at a previous hospital had shown no abnormal changes in the gastrointestinal mucosa. The patient had been taking 180 mg of loxoprofen sodium and 30 mg of lansoprazole every day for the past 3 years, for osteoarthritis of the knees, but had otherwise been healthy with no prior history of gastrointestinal disease. Family history was unremarkable.
On admission, weight was 53 kg and height was 144 cm. She was afebrile, with a blood pressure of 128/84 mmHg and a heart rate of 82 beats/min. The right lower abdominal quadrant was slightly painful on palpation, the face was swollen, and pretibial pitting edema was also present. No superficial lymph nodes were palpable. Laboratory investigations revealed: white blood cells, 7960/mm3; C-reactive protein, 0.31 mg/dL; total protein, 4.8 g/dL; plasma albumin, 2.8 mg/dL; no abnormalities in liver, renal, thyroid and adrenal function tests; negative serology for rheumatoid factor; normal results of urinalysis and no proteinuria. Moreover, no steatorrhea was identified, indicating that malabsorption syndrome was unlikely.
Endoscopic examination of the lower intestinal tracts showed edematous mucosa, diminished vascular transparency and multiple areas of erythema (Figure ). Multiple biopsies revealed changes consistent with collagenous colitis, showing prominent subepithelial eosinophilic band-like deposits with increased lymphocytes and plasma cells (Figure ). The distribution of the disease was whole colon and the findings were worse in the right compared to the left colon. The thickness of the collagen layer was 80-100 μm in the right and 40-50 μm in the left. There were no areas of cryptitis, crypt abscesses and no area of superimposed infection inflammatory bowel disease. Sloughing of surface epithelium was also identified. Endoscopic examination and biopsies of other portion of the gastrointestinal tract showed no obvious abnormalities in the stomach and the small intestine, and there was no evidence of celiac sprue. The duodenum showed normal villi and no obvious lymphocyte infiltration. Small bowel barium study showed no abnormalities. On 99mTc HSA scintigraphy, protein leakage was detected throughout the whole colon, particularly in the ascending portion (Figure ). No leakage was apparent from the stomach or small intestine. Neither chest/abdominal computed tomography (CT) nor 67Ga scintigraphy showed any evidence of malignancy. On the basis of these findings, collagenous colitis associated with PLE was diagnosed.
Colonoscopy shows an edematous mucosa, diminished vascular transparency throughout the colon on admission. Friability of mucosa and multiple erythema were observed (A). Endoscopic findings 1 mo after treatment showed normal mucosa (B).
Figure 2 Histology of the biopsied specimen demonstrates subepithelial from eosinophilic band-like deposit (arrows), with increased lymphocytes and plasma cells. Sloughing of surface epithelium is also shown (A). Epithelial detachment and inflammatory cells decreased, (more ...)
99mTc-human serum albumin scintigraphy shows leakage of the tracer in the large bowel (arrows) on admission (A) but no accumulation 1 mo after starting oral prednisolone (B).
After diagnosis, loxoprofen sodium and lansoprazole were discontinued. However, general condition remained unimproved. Administration of 1.8 g polycarbophil calcium, 2 g natural aluminum silicate, 2 g bifidobacterium, 20 mg scopolamine butylbromide, 4 mg loperamide hydrochloride and 2 g albumin tannate likewise did not improve symptoms, and serum protein and albumin levels remained low despite administration of human serum albumin. Frequency of diarrhea increased and abdominal pain became extremely severe, and the condition of the patient deteriorated. We decided to start prednisolone therapy after hypovolemic shock developed. Soon after initiating prednisolone treatment at 30 mg/d, abdominal pain and diarrhea dramatically improved along with general condition. Total plasma protein and plasma albumin levels gradually increased to 5.4 g/dL and 3.4 g/dL, respectively, by 1 mo after starting prednisolone therapy. Follow-up endoscopy showed normal-appearing colonic mucosa (Figure ), and multiple biopsies showed decreased epithelial detachment and inflammatory cells, although the collagen band beneath the mucosa was not reduced (Figure ). Additional 99mTc HSA scintigraphy showed no accumulation of tracer in the large bowel, indicating remission of PLE (Figure ).